Matsumoto Naoki, Osada Marie, Kaneko Kou, Ohara Ken, Noguchi Daito, Udagawa Haruhiko, Suzuki Nagazumi, Matsumoto Chieko, Takahashi Sachio
Department of Obstetrics and Gynecology, Fukaya Red Cross Hospital, 5-8-1 Kamishibachonishi, Fukaya City, Saitama 366-0052, Japan ; Department of Obstetrics and Gynecology, Tatedebari Sato Hospital, 96 Wakamatsucho, Takasaki City, Gunma 370-0836, Japan.
Case Rep Obstet Gynecol. 2013;2013:267268. doi: 10.1155/2013/267268. Epub 2013 Sep 8.
Pemphigoid gestationis (PG) is a rare, perinatal, autoimmune, and blistering dermatosis. Only few cases of PG involving hydatidiform moles have been reported. Complete hydatidiform moles are usually evacuated by dilatation and curettage. We report a patient with a massive complete hydatidiform mole that underwent spontaneous expulsion; she subsequently developed PG. A 19-year-old unmarried nulligravid woman was referred to our hospital following excessive vaginal bleeding after an uncertain amenorrheal period. The patient presented with preshock vital signs, severe anemia, and a positive urine pregnancy test. Imaging examinations revealed a massive intrauterine mass (19 × 15 × 10 cm), suggesting a complete hydatidiform mole. She was hospitalized and treated with blood transfusion. Sixteen hours after hospitalization, the massive molar mass underwent spontaneous expulsion and bleeding ceased. Three days after the expulsion, she developed pruritic skin lesions including papules, erythemas, and bullae, which spread over her entire body. Skin biopsy revealed PG and subepidermal blister formation and linear complement C3 deposition along the basement membrane zone, and the serum anti-BP180 antibody level was found to be high on measurement. She was effectively treated with 50 mg/day of oral prednisolone. Her skin lesions disappeared, leaving pigmentation.
妊娠类天疱疮(PG)是一种罕见的围产期自身免疫性水疱性皮肤病。仅有少数PG累及葡萄胎的病例报道。完全性葡萄胎通常通过扩张刮宫术清除。我们报告一例巨大完全性葡萄胎患者发生自然排出,随后发生了PG。一名19岁未婚未孕女性在经历一段不明停经后出现大量阴道出血,被转诊至我院。患者呈现休克前期生命体征、严重贫血,尿妊娠试验阳性。影像学检查显示子宫内有一巨大肿物(19×15×10cm),提示完全性葡萄胎。她住院并接受输血治疗。住院16小时后,巨大的葡萄胎块自然排出,出血停止。排出后三天,她出现瘙痒性皮肤损害,包括丘疹、红斑和大疱,遍布全身。皮肤活检显示为PG,有表皮下水疱形成,基底膜带可见线性补体C3沉积,检测发现血清抗BP180抗体水平升高。她接受了每日50mg口服泼尼松龙的有效治疗。她的皮肤损害消失,留有色素沉着。